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Title Burkholderia pickettii spondylitis
Author(s) Sudo, Hideki; Hisada, Y.; Ito, M.; Kotaki, H.; Minami, A.
Citation Spinal Cord, 43(8), 499-502https://doi.org/10.1038/sj.sc.3101740
Issue Date 2005-08
Doc URL http://hdl.handle.net/2115/14600
Rights Nature Publishing Group, Spinal Cord, vol. 43, 2005, 499-502
Type article
File Information SC2005-43-8.pdf
Hokkaido University Collection of Scholarly and Academic Papers : HUSCAP
Burkholderia Pickettii Spondylitis
Hideki Sudo, MD*
Yoshihisa Hisada, MD+
Manabu Ito, MD*
Hiroaki Kotaki, MD+
Akio Minami, MD*
*Department of Orthopaedic Surgery, Hokkaido University Graduate
School of Medicine, Sapporo, Japan; and + Department of Orthopaedic
Surgery, Nayoro City General Hospital, Nayoro, Japan
Correspondence;
Hideki Sudo, MD
Department of Orthopaedic Surgery
Hokkaido University Graduate School of Medicine
North-15, West-7, Kita-ku, Sapporo, Hokkaido 060-8638, Japan
Phone: +81-11-706-5934 FAX: +81-11-706-6054
E-mail: hidekisudo@yahoo.co.jp
Key words; Spondylitis, Burkholderia Pickettii
Running title; Burkholderia Pickettii Spondylitis
1
Abstract
Study design: Case report describing Burkholderia pickettii spondylitis in a
healthy adult.
Objectives: To describe this very rare form of spondylitis and to discuss
some of the difficulties in the diagnosis of Burkholderia pickettii
spondylitis.
Setting: Department of Orthopaedic Surgery, Nayoro City General Hospital,
Japan.
Methods: A 48-year-old woman presented with a complaint of severe back
pain radiating from the right side of her chest. Plain radiographs of the spine
showed osteolytic destruction of the right side of the T10 vertebral body at
T10 level, with an involvement of the pedicle. Magnetic resonance image of
the spine showed a low signal intensity from the T10 vertebral body on a
T1-weighted image and an increased signal intensity on T2-weighted
sequence image. These lesions were enhanced when a contrast medium was
used. The patient underwent open biopsy and specimens were collected
through the right pedicle.
Results: Diagnosis was established on the basis of direct identification of
the microorganism. Histological findings were consistent with examination
of Burkholderia Pickettii spondylitis. Chemotherapy (intravenous cefepime
and per os minocycline) resulted in complete cure.
2
Conclusion: B. Pickettii is widely distributed in aqueous sources in nature
and has not previously been considered to be an aggressive pathogen
towards humans. This case report will help to improve our understanding of
the ecology and virulent pathogenicity of this organism. A biopsy is an
essential and reliable method for the early etiologic diagnosis, which will
lead to prevent the development of more severe complications such as spinal
cord compression.
Key words; Spondylitis, Burkholderia Pickettii
3
Introduction
Burkholderia Pickettii is a nonfermentative gram-negative bacillus
isolated commonly from environmental sources and only rarely from
clinical samples.1 The species has very rarely been identified as a human
pathogen and has been associated with pseudobacteremias or in
asymptomatic colonization of patients.2,3 The cases in which infection has
been reported generally involve simple bacteremia, related to contamination
of manufactured products.4-6 The authors describe a rare case of B. Pickettii
spondylitis in a healthy adult.
4
Case report
A 48-year-old previously healthy woman was referred to our hospital in
March 2003 with a complaint of severe back pain radiating from the right
side of her chest. The pain had lasted for more than five weeks. Her past
history was unremarkable, except that a total hysterectomy had been
performed for hysteromyoma five years previously. On admission, the
patient’s temperature was 37.2 o C, her pulse rate was 66 beats / min, and her
blood pressure was 110/70 mm Hg. No skin lesions were observed. Physical
examination revealed tenderness on the spinous processes of the
thoraco-lumbar spine. No neurological other deficits were identified. The
peripheral white blood cell count was 5600 /mm3, and her C-reactive protein
level was 2.8 mg/dL. Other laboratory data including tumor markers were
within normal limits. Cultures of blood and urine yielded negative results.
Plain radiographs of the spine showed destruction of the right-side
vertebral body at T10 level, with an involvement of the pedicle (Figure1).
An axial image of the computed tomography scan at T10 level showed an
osteolytic area on the right side of the vertebra and pedicle (Figure2).
Magnetic resonance image (MRI) of the spine showed a low signal intensity
in the T10 vertebral body on a T1-weighted image (Figure3A) and a hyper-
intense signal on T2-weighted sequence image. These lesions were
enhanced with a contrast medium(Figure3B). A bone scan showed increased
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uptake in the T10 vertebra and 67Gallium scan showed pathological uptake
in the same lesion.
The patient underwent open biopsy and specimens were collected through
the right-side pedicle. Histopathological studies showed multiple foci of
polymorphonuclear cell infiltration. No malignant lesion was identified. All
cultures of samples obtained during biopsy yielded Burkholderia Pickettii.
The isolate was sensitive to cefotaxime, cefepime, minocycline, and
ciprofloxacin. The patient was treated with intravenous cefepime (1g / day)
for 2 weeks, followed by per os administration of minocycline (100mg /
day) for 3 months. The patient’s condition slowly improved and her back
pain subsided. At 18-month follow- up, the patient was free of symptoms.
All laboratory data were within normal limits. Plain radiographs did not
show further collapse of the vertebral body. MRI at T10 showed the same
intensity as other vertebral bodies.
6
Discussion
B. Pickettii is classified in the rRNA homology group II.7 It is widely
distributed in aqueous sources in nature and has not previously been
considered to be an aggressive pathogen towards humans.2,3 Cases in which
B. Pickettii infection has been reported, generally involve simple bacteremia
related to contamination of hospital pharmacy sources.4-6 Many cases were
associated with a direct inoculation of the blood or another site of infection.
Maki et al 4 reported an outbreak of nosocomial bacteremias caused by B.
Pickettii in surgical patients and the linked this to the first traces in a
contaminated narcotic. The epidemic was caused by fentanyl that became
contaminated in the Central Pharmacy of their hospital. The contamination
was introduced during convert removal of fentanyl from predrawn syringes
and replacement by an equal volume of contaminated distilled water.
Antiseptics such as chlorhexidine and ethacridine lactate have also been
reported to be contaminated with B. Pickettii.8,9 In addition to the
bacteremia, the organism has been reported to cause endocarditis and
meningitis.10,11 These previous reports described that most of the B. Pickettii
infection was associated with an immunocompromised state such as
haemodialysis or cancer therapy.10,11
Spondylitis caused by B. Pickettii is extremely rare. Most of the reported
cases of spondylitis or discitis are caused by more-virulent organisms.
7
Staphylococcus aureus is the more common pathogen, followed by
gram-negative bacteria and Streptococcus species.12 To our knowledge, only
one case of B. Pickettii spondylodiskitis has been reported. Wertheim et al 7
described a case of spondylodiskitis in a patient with chronic renal failure.
They emphasized that B. Pickettii might be a more virulent and invasive
pathogen than previously recognized. As to the portal or mechanism of B.
Pickettii entry, they suspected that the patient might have been infected
during a preceding period of hospitalization, by introduction of B. Pickettii
through an intravenous catheter or during cardiac catheterization. Moreover,
their patient’s situation was complicated by underlying diabetes, liver
disease, and uremia and by administration of corticosteroids, all of which
increased the risk of infection as previously described.10,11
In the present case, however, the patient was a previously healthy,
ordinary housewife. Pyogenic spondylodiskitis is uncommon in healthy
people. We believed that B. Pickettii was unlikely to be a contaminant
because the specimens were taken using a careful, sterile technique. B.
Pickettii was only isolated in pure culture from an intraoperative direct
inspection of the bone, and despite extensive investigation, no alternative
etiologic agent was identified. No cases of B. Pickettii infection were
uncovered in our hospital. The route of entry for the organism remains
unclear. Since blood cultures are negative in more than two-fifths of
8
spondylodiskitis, vertebra or disc-space aspiration biopsy offers an essential
and reliable method for the rapid etiologic diagnosis, like in this case.12
Although clinical findings in the present case were unremarkable
compared with other cases of pyogenic spondylitis, radiological findings
showed some distinctive features. Plain radiographs showed that the bony
destruction had spread over the pedicle. These images also demonstrated
that the structure of the vertebral endplates were relatively well maintained.
MRI at the infected vertebra exhibited a diffuse, homogeneous intensity
area. This lesion was enhanced when contrast medium was used. Intensity
changes in the intervertebral discs and vertebral endplates were not
observed. Intravertebral abscesses were not found. All these findings are
similar to metastases to the spine and can help aid the diagnosis of B.
Pickettii spondylitis.
In summary, we report the first case of spondylitis due to B. Pickettii in a
previously healthy adult, which will help us as physicians to improve our
understanding of the ecology and virulent pathogenicity of this organism.
9
References
1. Gilligan PH. Pseudomonas and Burkholderia. In: Murray PR, Baron EJ,
Pfaller MA, Tenover FC, Yolken RH,eds. Manual of clinical
microbiology, 6th ed. American Society for Microbiology, Washington,
DC.1995; 509-519.
2. Verschraegen G, Claeys G, Meeus G, et al. Pseudomonas pickettii as a
cause of pseudobacteremia. J Clin Microbiol. 1985;21:278-279.
3. McNeil MM, Solomon SL, Anderson RL, et al. Nosocomial
Pseudomonas pickettii colonization associated with a contaminated
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4. Maki DG, Klein BS, McCormick RD, et al. Nosocomial Pseudomonas
pickettii bacteremias traced to narcotic tampering. A case for selective
drug screening of health care personnel. JAMA. 1991; 265:981-986.
5. Fernandez C, Wilhelmi I, Andradas E, et al. Nosocomial outbreak of
Burkholderia pickettii infection due to a manufactured intravenous
product used in three hospitals. Clin Infect Dis. 1996; 22:1092-1095.
6. Chetoui H, Melin P, Struelens MJ, et al.Comparison of biotyping,
ribotyping, and pulsed-field gel electrophoresis for investigation of a
common-source outbreak of Burkholderia pickettii bacteremia. J Clin
Microbiol. 1997;35:1398-1403.
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7. Wertheim WA, Markovitz DM. Osteomyelitis and intervertebral discitis
caused by Pseudomonas pickettii. J Clin Microbiol. 1992;
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8. Kahan A, Philippon A, Paul G, et al. Nosocomial infections by
chlorhexidine solution contaminated with Pseudomonas pickettii
(Biovar VA-I). J Infect. 1983;7:256-263.
9. Oie S, Kamiya A. Bacterial contamination of commercially available
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10. Fass RJ, Barnishan J.Acute meningitis due to a Pseudomonas-like Group
Va-1 bacillus.Ann Intern Med. 1976;84:51-52.
11. Graber CD, Jervey LP, Ostrander WE, et al. Endocarditis due to a
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12. Jimenez-Mejias ME, de Dios Colmenero J, Sanchez-Lora FJ, et al.
Postoperative spondylodiskitis: etiology, clinical findings, prognosis,
and comparison with nonoperative pyogenic spondylodiskitis. Clin
Infect Dis. 1999;29:339-345.
11
Figure legend
Figure1
Plain radiographs of the spine showed destruction of the right-side vertebral
body at T10 level, with an involvement of the pedicle.
Figure2
An axial image of the computed tomography scan at T10 level showed an
osteolytic area on the right side of the vertebra and pedicle.
Figure3
MRI at T10 level demonstrated an osteolytic area on the right side of the
vertebra and pedicle, which exhibited a diffuse, homogeneous low intensity
on a T1-weighted image (Figure3A). These lesions were enhanced with a
contrast medium (Figure3B).
12
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